C L I N I C A L R E P O R T
A Rare Case Report of a Child Coexistence Thyroglossal Cyst and Second Branchial Cleft Fistulae
Rachid Mahdoufi1•Ismail Barhmi1•Nabil Tazi1•Sami Rouadi1•Reda Abada1• Mohamed Roubal1• Mohamed Mahtar1
Received: 29 October 2015 / Accepted: 10 May 2016 ÓAssociation of Otolaryngologists of India 2016
Abstract Thyroglossal duct cysts followed by branchial cleft anomalies are the most common congenital neck masses encountered in practice, second branchial cleft cysts and sinuses are the most common type (LaRiviere and Waldhausen in Surg Clin North Am 92(3):583–597, 2012). Although both abnormalities are common individ- ually, but rarely seen associated in same patient as described in our case. Congenitalcervical anomalies are important to consider in the differential of head and neck masses in children and adults. These lesions can present as palpable cystic masses, infected masses, draining sinuses, or fistulae. Thyroglossal duct cysts are most common, followed by branchial cleft anomalies. A synchronous presentation of both type of cyst and fistula in a same child patient is very rare with no such cases reported in literature till date.
Keywords Branchial fistulaThyroglossal cyst CoexistenceChild
Introduction
Thyroglossal duct cysts account for approximately 70 % of all congenital abnormalities of the neck. Anomalies of the second branchial fistula account for 90 % of the develop- mental abnormalities of the branchial apparatus. But a simultaneous presentation of second branchial cleft fistulae with thyroglossal cyst is a very rare presentation. We
present the first case report of thyroglossal cyst and left branchial sinus which appeared concurrently.
Case Report
An 8 year-old girl was admitted to our ENT department for recurrent, lateral cervical discharge from a fistula in the lower part of the neck since birth. She also complained of a lump on middle upper part of the neck for 6 month dura- tion which increased in size with time.
On physical examination, there was a discharging defect along the left anterior border of Sternocleidomastoid muscle at the junction of middle and upper 1/3rd of neck, an anterior palpable cervical mass next to the hyoid bone and it moves on swallowing and protrusion of the tongue.
However no active inflammation was present around the lesions. Oropharynx was normal with no loco regional lymphadenopathy. Also there was no sign of other con- genital anomalies in head neck region or elsewhere.
According to the above findings, our clinical diagnosis is thyroglossal cyst and chronic skin sinus left upper neck.
Ultrasound neck showed cystic lesion at the midline region of the neck likely of thyroglossal cyst. Sinus opening has medium tract which ends within the carotid bifurcation.
After necessary routine investigations, excision of the fistulous tract and thyroglossal cyst were carried out under general anesthesia.
During the operation, the thyroglossal cyst was removed completely together with part of the hyoid bone as a standard Sistrunk Operation. Excision of fistulous tract was done through elliptical one incision. Whole tract was removed, the upper blind end which blends with carotid bifurcation (Fig. 1). Post-operative period was uneventful and patient was discharged 1 day after.
& Ismail Barhmi
ismailbarhmi@gmail.com
1 ENT Department Face and Neck surgery, Hospital August 20, 1953, University Hospital Ibn Rochd, Casablanca, Morocco
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Indian J Otolaryngol Head Neck Surg DOI 10.1007/s12070-016-0995-z
Histology of both specimens confirmed the diagnosis of thyroglossal duct cyst (Fig.2) and branchial fistulae (Fig.3).
No recurrence of symptoms is seen with 6 months of follow-up.
Discussion
Thyroglossal duct cysts (TGDCs) followed by branchial cleft anomalies are the most common congenital neck masses encountered in practice, Second branchial cleft cysts and sinuses are the most common type [1]. Although both abnormalities are common individually, but rarely seen associated in same patient as described in our case.
TGDCs are the most common congenital midline neck masses that arise from a tubal remnant of thyroid descent
during development; it can be located anywhere along the course of the thyroglossal duct from the base of the tongue to the thyroid gland [2,3].
Patients present commonly with an asymptomatic, cys- tic neck mass in the midline near the hyoid bone, 66 % of the location for the cystic mass is adjacent to the hyoid bone [3,4]. About 75 % of patients with TGD anomalies are diagnosed before 30 years of age, and more than half of these are identified before age 10 years [2], same thing reported in this case.
Clinically TGDCs move upward on tongue protrusion and swallowing [5, 6]. However, restricted movement of the cyst does not rule out diagnosis. TGDCs may show similarities to dermoid cysts, lymphadenopathy, lymphatic malformations, ectopic thyroid gland, branchial cleft cysts, hemangiomas, lipomas, and sebaceous cysts [6, 7]. Thy- roglossal duct carcinomas occur in\1 % of TGDCs [8,9].
Ultrasonic Image (USI) is considered the method of choice for confirming clinical diagnosis of TDC and has an important role before surgery to rule out complications, and the lesion may be anechoic, homogeneously hypoechoic with intralesional septa, appear pseudo-solid due to possi- ble protein contents or heterogeneous [10].
The Sistrunk procedure is the surgical treatment of choice. The procedure includes excision of the cyst, the thyroglossal tract, and the central portion of the hyoid bone to prevent recurrence [1].
The epithelial lining of a TGDC ranges from squamous epithelium to pseudostratified ciliated columnar epithe- lium. Salivary gland tissue or thyroid gland tissue maybe seen in the wall of the cyst [11].
Branchial cleft anomalies may present as a cyst, sinus, fistula, or cartilaginous remnant [12]. Approximately 80 % Fig. 1 Intraoperative picture showing the coexistence of a thyroglos-
sal duct cyst (a) and fistula of the second branchial cleft (b)
Fig. 2 Thyroglossal duct histology
Fig. 3 Branchial fistulae histology
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of branchial cleft anomalies present as a cyst and about 95 % are formed from the region of the second branchial arch [13, 14]. Although branchial cleft fistulae are con- genital and might be noted at birth, most are not detected until the first or second decade of life.
Anatomically, a typical second cleft fistula has its external opening at the anterior border of sternomastoid at the junction of middle and lower 1/3rd. Second arch anomalies are classified into four types. Type I lesions lie anterior to the sternocleidomastoid muscle (SCM) and do not come in contact with the carotid sheath. Type II lesions are the most common and pass deep to the SCM and either anterior or posterior to the carotid sheath. Type III lesions pass between the internal and external carotid arteries and are adjacent to the pharynx. Type IV lesions lie medial to the carotid sheath close to the pharynx adjacent to the tonsillar fossa [15]. Our case had the type II pattern.
Clinically symptoms consist of intermittent or continu- ous mucous discharge from the anterior aspect of neck. In addition presence of infection may lead to formation of an abscess and signs of inflammation at the site of opening.
The diagnosis is established by physical examination and radiological investigations are rarely asked for. However, a fistulogram if performed delineates the tract and it is often the commonest investigation available [16].
The definite treatment of branchial anomalie is complete surgical excision of tract, most suitable age for surgery is 2–3 year or as early as possible if it is already delayed and among surgical techniques stepladder incision is most accepted method [17]. The reported incidence of recur- rence rate was 3 % where only external approach was used.
Conclusion
In conclusion, our case showed that there is a possibility that more than one anomaly can be seen in same patient simultaneously, so that we should always keep in mind and look for a possible associated or concurrent lesion.
Compliance with Ethical Standards Conflict of interest None.
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